GYNECOLOGIC
ONCOLOGY
39, 89-92
(1%)
CASE REPORT Gallstone lleus Masquerading as Recurrent Carcinoma of the Ovary M. PREFONTAINE,
M.D.,
FRCSC,*
M. HARDY, M.B.,
B.S., FRCSC,t
AND P. GENEST,
M.D.,
FRCPCS
*Department of Obstetrics ond Gynecology and TDepartment of Surgery, Ottawa General Hospital, and *Department of Radiation Ontario Cancer Foundation, Ottawa, Ontario KIH 8L.6, Canada
Therapy,
Received January 26, 1990 A case of gallstoneileti in a patient with carcinmna of the
ovary is presented.A 7%year-oldfemalewith stageIII Carcinoma of the ovary underwent opthiial debulking suige@followed by six coursesof chemotherapyand a microscopicallypositive second-look laparotomy. She w& trWed by whole-abdomenpelvic radiation. She then developedprogr&sive nausea,vomiting, ab+ dominaldistension,and eventually completesinall bowelobstruction. The diagnosisof gallstoneileus was made preoperativeiy basedon the radiological findings. The pathophysiolbgyof gallstone ileus is discussedin the differential diagnosisof patients treated for Earcinomaof the Ovary. 8 1990 AcsdetiRcas, I~C. INTRODUCTION
Despite known response to different chemotherapeutic agents and to radiation therapy the majority of patients with advanced carcinoma of the ovary suffer from recurrent disease. The most common site of failure is the peritoneal cavity, and most patients succumb to bowel obstruction and its complications. Radiation therapy at the doses prescribed for this malignancy may cause bowel obstruction if combined with multiple laparotomies. Women with carcinoma of the ovary presenting with a bowel obstruction are suspected of having recurrent disease, confirmed in most at laparotomy or by the clinical outcome. We report here a case of gallstone ileus masquerading as recurrent carcinoma of the ovary. The diagnosis was made preoperatively after a long period of intermittent symptomatology. A previous case of gallstone ileus in a patient with carcinoma of the ovary has been reported but not in the English literature (11. CASE PRESENTATION A 74-year-old woman presented in October 1986 with 6 months of increasing abdominal girth. She denied an-
orexia, nausea, vomiting, and changes in bowel or bladder function. She had a negative past medical history and functional inquiry, was on no medication, and had no known allergy. Her height and weight were respectively 153 cm and 50 kg. There was no peripheral adenopathy; chest and cardiovascular examinations were unremarkable. Her abdomen was markedly distended by ascites: no masses or visceromegaly were palpable. The vagina was stenotic, a large mass filled the pelvis and cul-de-sac. Preoperative chest X ray was normal, barium enema showed displacement of the sigmoid, and intravenous pyelogram showed displacement of the distal right ureter and a single large laminated gallstone in the right upper quadrant (Fig. 1). At surgery there were 5 liters of ascites and a 12 x U-cm mass arising from the tight ovary, encasing the appendix; the uterus and the left ovary were atrophic. A 2.5-cm gallstone was palpable in the gallbladder. Residual disease after total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy, and appendectomy consisted of diffuse peritoneal seedlings all less than 4 mm in diameter. Pathology showed a well-differentiated papillary serous cystadenocarcinoma of the ovary. At second-look laparotomy after six cycles of intravenous carboplatinum and cyclophosphamide there remained a single microscopic focus of carcinoma on the small bowel mesentery. She underwent irradiation: 2250 cGy to the whole abdomen and a boost of 2400 cGy to the pelvis. Between January 1988 and March 1989 she suffered recurrent borborygmi, crampy abdominal pain, nausea, vomiting, and alternate diarrhea and constipation. Her weight gradually decreased to 38 kg. Despite variably severe abdominal distension there was no palpable disease. Ultrasound and CT scan were negative and her CA-125 was normal. Loperamide, dietary manipulations,
89 009%8258&O $150 Copyright Q 1990 by Academic Press, Inc. All rights of reproduction in any form reserved.
90
PREFONTAINE,
HARDY,
AND GENEST
FIG. 1. Preoperative intravenous pyelogram in October 1986 showing a single large laminated gallstone in the right upper quadrant.
and supplementation brought temporary symptomatic improvement. She presented in March 1989 with worse vomiting, no flatus for 5 days, and dehydration. Two views of the abdomen showed a large laminated biliary calculus in the pelvis, distension of several small bowel loops, but no visible air in the biliary tree (Fig. 2). At laparotomy there were diffuse dense adhesions involving the small and large bowel. Radiation enteritis spared only a short segment of jejunum and the transverse colon. The gallstone was palpable through matted small bowel, impacted in the ileum, and was maneuvered back into a more proximal loop. Through a short enterotomy, a black nonfaceted 2.5-cm stone was extracted. The gallbladder, fibrosed to the duodenum, contained no other stone. It was elected not to perform a cholecystectomy. There was no evidence of carcinoma and biopsies from a few adhesions were negative for carcinoma. She had a pro-
longed ileus and remained on intravenous hyperalimentation until her discharge 6 weeks after surgery. She was readmitted in June 1989 with persistent subocelusion signs and symptoms. Antegrade small bowel enema showed very slow transit but no focal obstruction. Metoclopramide, domperidome, and cisapride did not improve her motility. She refused another attempt at surgical correction of her chronic enteritis and occluded both subclavian veins, through which she was receiving total parenteral nutrition. She was discharged home at her request and that of her family in September and expired at home on November 1, 1989. No autopsy was performed. DISCUSSION In women treated for stage III and IV ovarian carcinoma presenting with bowel obstruction the differential
CASE REPORT
FIG.
91
2. Supine abdominal radiograph showing distended small bowel loops and a large gallstone in the pelvis c:onsistent with a gallstone
diagnosis includes recurrent ovarian carcinoma, surgical adhesions, a second primary, and, when applicable, radiation enteritis. When obstructed most patients with recurrent carcinoma have ascites or palpable disease. Our patient developed borborygmi, abdominal pain, and distension 8 months following second-look laparotomy. Over the next 15 months she had increasingly frequent and severe subocclusive symptoms. Despite persistent abdominal distension there was no clinical, radiological or biochemical evidence of disease. We suspected occult recurrence; the lack of objective findings and heavy pretreatment favored no active therapy. Rates of intestinal morbidity requiring surgical treatment of 2 to 18% after whole-abdomen and pelvic radiation suggested this etiology [2-51. Prior to complete obstruction plain films of the abdomen were not done. When our patient developed a complete obstruction, gallstone ileus was diagnosed by abdominal radiographs
and confirmed at surgery. Gallstone ileus, a mechanical obstruction of the intestine caused by impaction of a gallstone in the lumen of the bowel, was first reported by Bartholin in the 17th century. It affects the elderly with a ratio of 10 females to 1 male. Pathophysiologically, a biliary enteric fistula forms from chronic pressure necrosis and erosion into adjacent bowel by a large stone, or severe acute cholecystitis progresses to a localized gangrenous perforation into adherent bowel, most often the duodenum. Without distal biliary obstruction the fistula may close spontaneously. A gallstone that reaches the gastrointestinal tract may be vomited or may pass the ileocecal valve without causing obstruction; most obstructions result from stones larger than 2.5 cm. The obstruction is initially incomplete with intermittent impaction and disimpaction of the stone usually in the distal ileum. Symptoms associated with gallstone ileus are abdom-
92
PREFONTAINE,
HARDY, AND GENEST
inal pain, vomiting, constipation, anorexia, and abdominal distension. The interval from onset of symptoms to admission varies from 12 hr to 1 month. Physical examination is consistent with small bowel obstruction. Our patient exhibited only two of the radiological signs of gallstone ileus: pneumobilia or air in the biliary tree, radiological evidence of partial or complete intestinal obstruction, visualization of the gallstone in the bowel lumen, and a change in position of a previously known gallstone. The diagnosis is rarely made preoperatively despite the presence of radiological signs. The obstruction usually occurs as an emergency in elderly patients who are often debilitated and dehydrated. Following rehydration one can attempt at laparotomy to milk the stone into the colon but more often it is removed by proximal enterotomy away from the obstruction, and a search is made for other stones to prevent recurrence. The indication for cholecystectomy and closure of the fistula, preferably postponed, remains controversial thereafter. Postoperative morbidity and mortality are high [6,7]. It is difficult to estimate the duration of intermittent gallstone impaction and disimpaction as the cause of our patient’s symptoms. The adhesions from her two prior laparotomies and irradiation undoubtedly increased the difficulty in removing the stone and led to persistent subocclusion afterward and to her eventual demise, if not to the constellation of symptoms prior to the diagnosis of gallstone ileus. Though uncommon, gallstone ileus should be considered in the differential diagnosis of an elderly female with bowel obstruction and known cholelithiasis. No conclusion can be drawn from this
single case. However, since the rate of complication from incidental cholecystectomy with major gynecologic cancer surgery is minimal, it should be considered in those cases of solitary large stones where abdominal irradiation is anticipated (81. REFERENCES
2.
3.
4.
5.
Jankowska, E., and Tyszko, B. Mechanical obstruction caused by a biliary stone in a patient with bilateral huge ovarian tumor. Wind Lek. 19(S), 413-414 (1966). Dembo, A. J., Bush, R. S., Beale, F. A., Bean, H. A., Pringle, J. F., Sturgeon, .I., and Reid, J. G. Ovarian carcinoma: Improved survival following abdominopelvic irradiation in patients with a complete pelvic operation. Amer. J. Obsfet. Gynecol. 134(7), 793-800 (1979). Hainsworth, J. D., Malcolm, A., Johnson, D. H., Burnett, L. S., Jone, H. W., and Greco, F. A. Advanced minimal residual ovarian carcinoma: Abdominopelvic irradiation following combination chemotherapy. Obstet. Gynecol. 61(5), 619-623 (1983). Tak, W. K., Costanza, M. E., Marchant, D. J., Munzenrider, J. E., Mitchell, G. W., Jr., Nathanson, L., and Emami, B. Surgery, chemotherapy and supervoltage radiotherapy for carcinoma of the ovary. Radiat. Oncol. Biol. Phys. 2(9), 895-901 (1977). Shelley, W. E., Starreveld, A. A., Carmichael, J. A., O’Connell, G., Roy, M., and Swenerton, K. Toxicity of abdominopelvic radiation in advanced ovarian carcinoma patients after cisplatin/cyclophosphamide therapy and second-look laparotomy. Obstet. Gynecul. 71(3), 327-332 (1988). Sabiston, D. C. Essentials of surgery, W. B. Saunders, Philadelphia, Chap. 28 (1987). Day, E. A., and Marks, C. Gallstone ileus: Review of the literature and presentation of thirty-four new cases. Amer. J. Surg. 129, 552558 (1975). Patsner, B., Mann, W. J., Arato, M., Maderiega, J., and Frei, L. W. Cholecystectomy accompanying major abdominal surgery for gynecologic cancer. Gynecol. Oncol. 32, 46-48 (1989).
ONCOLOGY
39, 89-92
(1%)
CASE REPORT Gallstone lleus Masquerading as Recurrent Carcinoma of the Ovary M. PREFONTAINE,
M.D.,
FRCSC,*
M. HARDY, M.B.,
B.S., FRCSC,t
AND P. GENEST,
M.D.,
FRCPCS
*Department of Obstetrics ond Gynecology and TDepartment of Surgery, Ottawa General Hospital, and *Department of Radiation Ontario Cancer Foundation, Ottawa, Ontario KIH 8L.6, Canada
Therapy,
Received January 26, 1990 A case of gallstoneileti in a patient with carcinmna of the
ovary is presented.A 7%year-oldfemalewith stageIII Carcinoma of the ovary underwent opthiial debulking suige@followed by six coursesof chemotherapyand a microscopicallypositive second-look laparotomy. She w& trWed by whole-abdomenpelvic radiation. She then developedprogr&sive nausea,vomiting, ab+ dominaldistension,and eventually completesinall bowelobstruction. The diagnosisof gallstoneileus was made preoperativeiy basedon the radiological findings. The pathophysiolbgyof gallstone ileus is discussedin the differential diagnosisof patients treated for Earcinomaof the Ovary. 8 1990 AcsdetiRcas, I~C. INTRODUCTION
Despite known response to different chemotherapeutic agents and to radiation therapy the majority of patients with advanced carcinoma of the ovary suffer from recurrent disease. The most common site of failure is the peritoneal cavity, and most patients succumb to bowel obstruction and its complications. Radiation therapy at the doses prescribed for this malignancy may cause bowel obstruction if combined with multiple laparotomies. Women with carcinoma of the ovary presenting with a bowel obstruction are suspected of having recurrent disease, confirmed in most at laparotomy or by the clinical outcome. We report here a case of gallstone ileus masquerading as recurrent carcinoma of the ovary. The diagnosis was made preoperatively after a long period of intermittent symptomatology. A previous case of gallstone ileus in a patient with carcinoma of the ovary has been reported but not in the English literature (11. CASE PRESENTATION A 74-year-old woman presented in October 1986 with 6 months of increasing abdominal girth. She denied an-
orexia, nausea, vomiting, and changes in bowel or bladder function. She had a negative past medical history and functional inquiry, was on no medication, and had no known allergy. Her height and weight were respectively 153 cm and 50 kg. There was no peripheral adenopathy; chest and cardiovascular examinations were unremarkable. Her abdomen was markedly distended by ascites: no masses or visceromegaly were palpable. The vagina was stenotic, a large mass filled the pelvis and cul-de-sac. Preoperative chest X ray was normal, barium enema showed displacement of the sigmoid, and intravenous pyelogram showed displacement of the distal right ureter and a single large laminated gallstone in the right upper quadrant (Fig. 1). At surgery there were 5 liters of ascites and a 12 x U-cm mass arising from the tight ovary, encasing the appendix; the uterus and the left ovary were atrophic. A 2.5-cm gallstone was palpable in the gallbladder. Residual disease after total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy, and appendectomy consisted of diffuse peritoneal seedlings all less than 4 mm in diameter. Pathology showed a well-differentiated papillary serous cystadenocarcinoma of the ovary. At second-look laparotomy after six cycles of intravenous carboplatinum and cyclophosphamide there remained a single microscopic focus of carcinoma on the small bowel mesentery. She underwent irradiation: 2250 cGy to the whole abdomen and a boost of 2400 cGy to the pelvis. Between January 1988 and March 1989 she suffered recurrent borborygmi, crampy abdominal pain, nausea, vomiting, and alternate diarrhea and constipation. Her weight gradually decreased to 38 kg. Despite variably severe abdominal distension there was no palpable disease. Ultrasound and CT scan were negative and her CA-125 was normal. Loperamide, dietary manipulations,
89 009%8258&O $150 Copyright Q 1990 by Academic Press, Inc. All rights of reproduction in any form reserved.
90
PREFONTAINE,
HARDY,
AND GENEST
FIG. 1. Preoperative intravenous pyelogram in October 1986 showing a single large laminated gallstone in the right upper quadrant.
and supplementation brought temporary symptomatic improvement. She presented in March 1989 with worse vomiting, no flatus for 5 days, and dehydration. Two views of the abdomen showed a large laminated biliary calculus in the pelvis, distension of several small bowel loops, but no visible air in the biliary tree (Fig. 2). At laparotomy there were diffuse dense adhesions involving the small and large bowel. Radiation enteritis spared only a short segment of jejunum and the transverse colon. The gallstone was palpable through matted small bowel, impacted in the ileum, and was maneuvered back into a more proximal loop. Through a short enterotomy, a black nonfaceted 2.5-cm stone was extracted. The gallbladder, fibrosed to the duodenum, contained no other stone. It was elected not to perform a cholecystectomy. There was no evidence of carcinoma and biopsies from a few adhesions were negative for carcinoma. She had a pro-
longed ileus and remained on intravenous hyperalimentation until her discharge 6 weeks after surgery. She was readmitted in June 1989 with persistent subocelusion signs and symptoms. Antegrade small bowel enema showed very slow transit but no focal obstruction. Metoclopramide, domperidome, and cisapride did not improve her motility. She refused another attempt at surgical correction of her chronic enteritis and occluded both subclavian veins, through which she was receiving total parenteral nutrition. She was discharged home at her request and that of her family in September and expired at home on November 1, 1989. No autopsy was performed. DISCUSSION In women treated for stage III and IV ovarian carcinoma presenting with bowel obstruction the differential
CASE REPORT
FIG.
91
2. Supine abdominal radiograph showing distended small bowel loops and a large gallstone in the pelvis c:onsistent with a gallstone
diagnosis includes recurrent ovarian carcinoma, surgical adhesions, a second primary, and, when applicable, radiation enteritis. When obstructed most patients with recurrent carcinoma have ascites or palpable disease. Our patient developed borborygmi, abdominal pain, and distension 8 months following second-look laparotomy. Over the next 15 months she had increasingly frequent and severe subocclusive symptoms. Despite persistent abdominal distension there was no clinical, radiological or biochemical evidence of disease. We suspected occult recurrence; the lack of objective findings and heavy pretreatment favored no active therapy. Rates of intestinal morbidity requiring surgical treatment of 2 to 18% after whole-abdomen and pelvic radiation suggested this etiology [2-51. Prior to complete obstruction plain films of the abdomen were not done. When our patient developed a complete obstruction, gallstone ileus was diagnosed by abdominal radiographs
and confirmed at surgery. Gallstone ileus, a mechanical obstruction of the intestine caused by impaction of a gallstone in the lumen of the bowel, was first reported by Bartholin in the 17th century. It affects the elderly with a ratio of 10 females to 1 male. Pathophysiologically, a biliary enteric fistula forms from chronic pressure necrosis and erosion into adjacent bowel by a large stone, or severe acute cholecystitis progresses to a localized gangrenous perforation into adherent bowel, most often the duodenum. Without distal biliary obstruction the fistula may close spontaneously. A gallstone that reaches the gastrointestinal tract may be vomited or may pass the ileocecal valve without causing obstruction; most obstructions result from stones larger than 2.5 cm. The obstruction is initially incomplete with intermittent impaction and disimpaction of the stone usually in the distal ileum. Symptoms associated with gallstone ileus are abdom-
92
PREFONTAINE,
HARDY, AND GENEST
inal pain, vomiting, constipation, anorexia, and abdominal distension. The interval from onset of symptoms to admission varies from 12 hr to 1 month. Physical examination is consistent with small bowel obstruction. Our patient exhibited only two of the radiological signs of gallstone ileus: pneumobilia or air in the biliary tree, radiological evidence of partial or complete intestinal obstruction, visualization of the gallstone in the bowel lumen, and a change in position of a previously known gallstone. The diagnosis is rarely made preoperatively despite the presence of radiological signs. The obstruction usually occurs as an emergency in elderly patients who are often debilitated and dehydrated. Following rehydration one can attempt at laparotomy to milk the stone into the colon but more often it is removed by proximal enterotomy away from the obstruction, and a search is made for other stones to prevent recurrence. The indication for cholecystectomy and closure of the fistula, preferably postponed, remains controversial thereafter. Postoperative morbidity and mortality are high [6,7]. It is difficult to estimate the duration of intermittent gallstone impaction and disimpaction as the cause of our patient’s symptoms. The adhesions from her two prior laparotomies and irradiation undoubtedly increased the difficulty in removing the stone and led to persistent subocclusion afterward and to her eventual demise, if not to the constellation of symptoms prior to the diagnosis of gallstone ileus. Though uncommon, gallstone ileus should be considered in the differential diagnosis of an elderly female with bowel obstruction and known cholelithiasis. No conclusion can be drawn from this
single case. However, since the rate of complication from incidental cholecystectomy with major gynecologic cancer surgery is minimal, it should be considered in those cases of solitary large stones where abdominal irradiation is anticipated (81. REFERENCES
2.
3.
4.
5.
Jankowska, E., and Tyszko, B. Mechanical obstruction caused by a biliary stone in a patient with bilateral huge ovarian tumor. Wind Lek. 19(S), 413-414 (1966). Dembo, A. J., Bush, R. S., Beale, F. A., Bean, H. A., Pringle, J. F., Sturgeon, .I., and Reid, J. G. Ovarian carcinoma: Improved survival following abdominopelvic irradiation in patients with a complete pelvic operation. Amer. J. Obsfet. Gynecol. 134(7), 793-800 (1979). Hainsworth, J. D., Malcolm, A., Johnson, D. H., Burnett, L. S., Jone, H. W., and Greco, F. A. Advanced minimal residual ovarian carcinoma: Abdominopelvic irradiation following combination chemotherapy. Obstet. Gynecol. 61(5), 619-623 (1983). Tak, W. K., Costanza, M. E., Marchant, D. J., Munzenrider, J. E., Mitchell, G. W., Jr., Nathanson, L., and Emami, B. Surgery, chemotherapy and supervoltage radiotherapy for carcinoma of the ovary. Radiat. Oncol. Biol. Phys. 2(9), 895-901 (1977). Shelley, W. E., Starreveld, A. A., Carmichael, J. A., O’Connell, G., Roy, M., and Swenerton, K. Toxicity of abdominopelvic radiation in advanced ovarian carcinoma patients after cisplatin/cyclophosphamide therapy and second-look laparotomy. Obstet. Gynecul. 71(3), 327-332 (1988). Sabiston, D. C. Essentials of surgery, W. B. Saunders, Philadelphia, Chap. 28 (1987). Day, E. A., and Marks, C. Gallstone ileus: Review of the literature and presentation of thirty-four new cases. Amer. J. Surg. 129, 552558 (1975). Patsner, B., Mann, W. J., Arato, M., Maderiega, J., and Frei, L. W. Cholecystectomy accompanying major abdominal surgery for gynecologic cancer. Gynecol. Oncol. 32, 46-48 (1989).
